Monday, March 05, 2018

We had agreed to be interviewed for this article. We felt it was an opportunity to inform Spectrum's readers about the scope and importance of the roles autistic people may play in autism research.

Instead, to our disappointment, the two
named autistic people in the article were subject to misrepresentation and a
systematic exclusion of their voices.

Unlike all others named and featured in the
article, neither Kabie Brook nor Michelle Dawson was interviewed, neither was directly
quoted, and neither was provided any links to their work or online profiles.

The message is that autism research is not
something these autistics can communicate about, much less contribute to as
equals, and that autistics must always and only be spoken for by others.

The striking segregation of named autistic
people in this article is inconsistent with journalist ethics. The autistics
who were misrepresented and denied a voice have been directly harmed.

Our goal is to bring this to the attention
of Spectrum readers, so they may be better informed. We hope that Spectrum will
improve its standards in the future.

Friday, November 10, 2017

In 2015 Autistica invited me to write a short blog post on lessons from history for autism research. Then they took down their entire blog (was it something I said?). What I wrote remains relevant and then some, so here it is again. There are two minor wording changes. The original (archived here) had no links; I've added a token few of the countless possible.

History tells us about the great importance of standards in human research—standards of science, standards of ethics. History also tells us that segregation harms human beings. The costs and injustices of segregated schools, workplaces, and neighborhoods are well known, but there are other more insidious ways to segregate. Denying a group of people the benefit and protection of standards in research is especially harmful and violates basic human rights. It is segregation at its worst, destructive to human well-being and scientific progress both. Yet in autism intervention research, these lessons have not been learned.

Autistics have long been subject to separate, unequal standards in both research and practice. Claims that standards must be lower for autistics than would be acceptable for anyone else continue to prevail. When it comes to autism interventions, autism-specific low standards have proliferated, by popular demand, and are now nearly universal. History tells us to recognize this as segregation at its insidious worst, but this has not yet happened.

Instead autism organizations and advocacy leaders, including those known for opposing each other, agree when it comes to segregating autistics this way. In their documents, policies, recommendations, and lobbying, they all promote autism-specific low standards—especially in interventions. Ignoring the strong lessons of history, they all support the segregation of autistics from the basic rights and standards which protect and benefit everyone else. Their high-profile disputes are, instead, over which kinds of bad science and bad ethics should be foisted on autistics.

The results are obvious. In non-autism areas, poor quality research and its harms—its waste of resources, its misleading findings—are vigorously condemned. There is recognition that even the best existing research standards are flawed and need always to be improved. But when it comes to autism, standards have instead been lowered or discarded to accommodate the extremely poor autism intervention literature. Poor standards in intervention research are seen not as harmful and wasteful, which they are, but as what autistics need and deserve. Resources have poured not into improving these abysmal standards, but into making the very poor quality autism intervention literature more powerful and influential.

In one influential paper, the authors devised an elaborate system of autism-specific low standards for evaluating intervention research, then suggested extending this segregated system to other developmentally disabled individuals. The authors did not suggest doing this to the typical population, to people like themselves—this clearly would be harmful and unacceptable.

The segregation of autistics from basic standards in research has lowered the bar for standards applied to autistics in other areas: in practice and professional ethics, in public policy, politics, law, journalism, medicine, education, and the list goes on. It has further removed any incentive to fund, conduct, or publish good quality autism intervention research. Yet, the popular effort to impose autism-specific low standards in the area of intervention research can be shown to lack any scientific or ethical foundation.

It is remarkable that while alarming claims about “costs of autism” are everywhere, there has been no concern whatsoever about the high costs, at every level, of autism-specific low standards. What is the way forward? We should listen to what history tells us. We should stop segregating autistics from the basic rights and standards everyone else can take for granted. Then we can see what else is needed.

Wednesday, December 02, 2015

In the Carter decision, the Supreme Court of Canada struck down the existing prohibition against physician-assisted dying, and gave the Canadian government until February 2016 to craft a legislative response. In response, an expert panel was created; see more information about the panel, Carter, and assisted dying here. Very late in the panel's consultations, I was asked to provide comments about assisted dying and autism. My very brief submission, possibly too late to be considered, is below, along with a few further notes.

My purpose is not to take any position on the issue of physician-assisted dying, but to call attention to the situation of autistic adults in Canada with respect to this issue.

Autistic adults will be affected by laws on physician-assisted dying in as many ways and in as many roles as will nonautistic adults. For example, autistic adults may be patients or physicians and as such may variously wish to obtain, to avoid, to provide, or not to provide assisted death.

What sets autistic adults apart is their difficult situation in Canada when it comes to basic scientific, medical, and ethical standards—standards which are often described as universal. These are the standards on which the Supreme Court of Canada (SCC) Carter decision implicitly and explicitly depends. The decision’s premise is that these standards will indeed benefit and protect everyone, whatever their role or wishes with regard to assisted dying.

While this premise is sound for the nonautistic population, for autistics in Canada it fails and must be rejected. Autistics have long been denied the “universal” basic standards on which Carter depends. There is a long history of scientific and ethical standards being lowered or discarded in autism research, a long history of unfounded but dominant claims that basic standards are somehow bad for autistics, or are too difficult to apply, or are not merited by this population. Lower standards for autistics than would be acceptable for anyone else have spread well beyond research and now permeate autism practice, advocacy, jurisprudence, journalism, policy, and politics.

In Canada there is no organization which promotes basic scientific, medical, and ethical standards for autistics. To the contrary, any suggestion that standards for autistics should resemble those for everyone else has been strongly opposed. This is reflected in public policy and generally in how autistics are regarded and treated in every aspect of their lives.

Under the dominant low standards, autism is viewed as grievous, as irremediable (beyond a tiny early window of opportunity), as causing enduring intolerable suffering, as an appalling burden on families and society. Media stories have mentioned individuals in Belgium euthanized for autism (Lerner & Caplan, 2015). A recent paper, while very preliminary, found that autistic adults seeking assisted dying are not mere rumors (Theinpont et al., 2015).

What the SCC’s Carter decision means with respect to autism is unclear. We are not in Belgium’s “very different medico-legal culture.” But in Canada autistics are subject to “very different” and profoundly lower standards than the court envisioned. This has consequences for autistics even if autism itself is not or does not become a condition for which assisted dying is granted.

Adults diagnosed as autistic disproportionately experience suicide, suicidality, and premature death (Lai & Baron-Cohen, 2015; Hirvikoski et al., 2015), plausibly as the predictable outcomes of being subject to unacceptably low standards. It remains to be seen how enduring intolerable suffering will be assessed and treated in these circumstances, which the SCC did not envision. The obvious solution, the only one consistent with the Charter, is to accord to autistics the universal scientific, medical, and ethical standards they are currently denied. So long as this does not happen, autistics in Canada do not fall under the overarching premise of the Carter decision. In all aspects of assisted dying, they will face hazardous, onerous, and unacceptable disadvantages.

Submitted
to: External Panel on Options for a Legislative
Response to Carter v. Canada, November 23, 2015, by Michelle
Dawson, CETEDUM, University of Montreal

Further notes
For more about autistics being denied basic standards in research, and some consequences of this, see my recent guest post for Autistica on history, segregation, and autism. For the current complicated situation regarding assisted dying in Quebec, see recent update.

Postscript: On Dec 3, the Canadian government asked the SCC for an additional six months to respond to Carter. For further updates, see the comments.

Was research something you always felt you had been quite good at, statistics, numbers etc? And what support network did you have to start this job?

Michelle Dawson:

No, I ran into research by accident--as I was trying to sort out legal issues. Then suddenly I had information I could really work with. Before that I had been totally discouraged from even looking at research--I was told that autistics could not understand research.

I haven't really had support, I just leveled a lot of criticism at a research group I took seriously and they offered me an affiliation.

Peter:

Hi Michelle, really interesting to hear your answers to the last questions. What sort of advice would you give to autistic people here in the UK who would like to get into research?

Michelle Dawson:

My main advice is--read a lot of papers! Find out what you're interested in and what you can do well. You need a lot of critical thinking and it helps to not decide ahead of time what you should find.

I also suggest reading the scientific literature involving the typical population to get an idea of the often higher standards there--and then use these higher standards to interpret the autism literature.

It is hard for me to give advice at the usual levels because I haven't ever been to university as a student. Still I hope I've shown that even an uneducated autistic can be useful in research, given the opportunity.

Kate:

What do you think about the links between research and government policy? I think we struggle to get government to fund support and interventions for children partly because they're not convinced of the benefits. What should we do about that?

Michelle Dawson:

The problem has been the poor quality of autism intervention research, resulting in arguments based on a few studies of questionable quality. In my view, the solution is to ensure that only good quality--as this is defined for the typical population--intervention research is funded.

We should at this point know much more than we do, about how to help and how not to harm autistics. We should have 10 or more large, well-designed, multi-site RCTs like the one Jonathan Green has conducted in the UK. But we don't. So policy and so on is determined according to who is loudest and so on, rather than according to what might help autistics.

Joe:

If you had the power to fund one new area of research what would it be and why?

Michelle Dawson:

I've been asked that question many times in the UK and my answer is always that I would throw funding into Jonathan Green's work, simply because he is applying much higher standards to autistics in autism intervention research than has previously been the case.

This may seem like an odd choice, but the poor quality of the autism intervention literature is in my view a major problem that doesn't stay confined to how decisions are made about interventions, which is important enough in itself.

We don't know yet how far better quality research will go in making things better for autistics, and to what degree it will solve the problems so to speak, but it is something we must try. The effort to lower standards for autistics has been dominant and it is hard to imagine a better way to signal that autistics are worth less than nonautistics.

It is very fortunate that Jonathan Green and others, like Catherine Aldred, have not gone along with this trend, and instead are viewing autistics as deserving of the same standards as everyone else. And we know that these standards work for autistics. So this is why I would send them more funding if I could.

Peter:

How do we support more autistics to become leading academics / scientists rather than purely the subject of research? What needs to change and are there any examples of where you think this is working well?

Michelle Dawson:

First I think we have to stop discouraging autistics from being interested in research. I've been discouraged by pretty much every major advocacy group, for example, on every political "side."

Also, there has been a tendency to segregate autistics from science as usual--of saying that science doesn't really work on autistics so we must have some special model or we must have autism-specific standards.

In my view, there is no evidence to support this longstanding trend--instead the problem has been that via advocacy, autistics have been segregated from the standards and so on that we know must exist to protect and benefit nonautistics.

So currently there is the idea that while autistics can participate in research, we can only do so under certain models, which are, for example, far too sophisticated and elaborate for me.

The group in Montreal seems to work well because there is no such elaborate model. We just do science, and this is something that some autistics are really good at. Autistics and nonautistics work together as equals and it is no big deal. There is no need to adhere to some elaborate ideological model about how autistics and academics should work together.

Jean-Paul:

Do you think autism researchers and autism advocates could and should worker closer together?

Michelle Dawson:

Since 1970 or before, advocacy (of various kinds) has been decisive in influencing the course of autism research. The question is whether this has been good for autistics. In many cases, I believe it hasn't been.

In part the responsibility lies on researchers who have used advocacy to prop up less than scientific (and less than ethical) views, theories, and practices.

I think it's very important that scientific and ethical standards not be lowered or discarded for autistics, and sadly there are a lot of demands via advocacy (from every direction) that this must happen.

I am really hoping for advocacy which demands higher standards of science and ethics for autistics, but this hasn't happened yet. Feel free to step in!

Sammi:

Do you think the politics of the autism community gets in the way of progress sometimes?

Michelle Dawson:

I think there is very strong evidence of how autism politics (on every "side") has harmed autistics.

The political process and political methods are important and they work, even if they look messy, but in autism they have dominated at the expense of scientific and ethical standards, and this has in my view harmed autistics, as it would for any other group.

There is more at the link. The photo above was taken by Thomas Henderson on an iPad, also the one below, both in Manchester. A lot of London, including the taxis, looked familiar due to Stephen Wiltshire's work. My experience in the UK was excellent. That was entirely due to those many who bravely welcomed and helped me out, who were extraordinarily forgiving of my many limitations, and who provided a lot of productive discussion.

From 1999, Pam Heaton again, as well as Linda Pring, Beate Hermelin, and others:

The term "Idiot-Savants" was first used by Langdon Down in 1887

Darold Treffert, often described as the authority on savants, has written accounts along these lines:

However, the first specific description of savant syndrome took place in London in 1887 when Dr J. Langdon Down gave that year’s prestigious Lettsomian Lecture at the invitation of the Medical Society of London... In 1887, ‘idiot’ was an accepted classification for persons with an IQ below 25, and ‘savant’, or ‘knowledgeable person’, was derived from the French word savoir meaning ‘to know’. Down joined those words together and coined the term idiot savant by which the condition was generally known over the next century.

So far as I can tell, this consensus is wrong. Edouard Seguin, who died in 1880, is well known for having written about savants. He wrote about the famous pianist Blind Tom Wiggins, for instance, in a book published in 1866. And in a short 1870 paper, he is quoted as using the term "idiot savant." Here it is (spelling from original):

Among the wealthier classes, idiocy is not only oftener aggravated by accessory diseases, but also complicated with abnormal semi-capacities or disordered instincts, which produce heterogeneous types to an almost unlimited extent. It is from this class, almost exclusively, that we have musical, mathematical, architectural, and other varieties of the idiot savant; the useless protrusion of a single faculty, accompanied by a woful general impotence.

Seguin's use of "idiot savant" did not pass unnoticed in the literature. For example, in the BMJ in 1875, George W. Grabham quotes and takes issue with Seguin's views (spelling from the original):

A curious class may be termed that of the idiot "savans", in whom one or more faculties are amazingly developed, perhaps to the detriment of the rest. One has a marvellous power of acquiring languages and musical knowledge; another, great mechanical skill and original constructive ability; a third, though very childish, is no mean mental arithmetician; a fourth remembers all he reads; a fifth delights in dates; while a sixth can tell the time when awakened from sleep. General improvement has taken place in all these cases.

Dr. Seguin, a well known authority on idiocy, has given the support of his pen to a theory "that idiocy is found in its simplest forms among the labouring classes, and that, among the wealthier classes, it is not only oftener aggravated by accessory diseases, but also complicated with abnormal semi-capacities or disordered instincts, which produce heterogeneous types to an almost unlimited extent. It is from this class almost exclusively that we have musical, mathematical, architectural, and other varieties of the idiot savant; useless protrusion of a single faculty, accompanied by a woeful general impotence". I am quite unable to agree with this view; my experience of many of these idiot "savans" proving them to have sprung from parents in humble circumstances, and leading me to believe them to have resulted in many instances from hereditary insanity.

It's possible Seguin was not the first to use "idiot savant" but he does get this honor in the online OED, which quotes Seguin's 1870 paper but does not mention or quote Langdon Down.

In a footnote, Spitz (1995) provides a small trace of dissent, noting that Down himself made no claim, in 1887, to having coined "idiot savant" and indeed he seems to be using an existing term. Spitz did not try to find who did coin "idiot savant," but you can't blame Down for the false consensus.

And it hardly matters, to current-day autistics, who exactly coined an obsolete term in the 1800s. Langdon Down and Edouard Seguin probably don't care about their h-indexes. There are many vastly more important issues related to the term "idiot savant" and the human beings who were characterized this way, and the still-dominant view that strong autistic abilities are useless protrusions (recent example here).

But it does matter when telling an inaccurate story becomes the standard in the autism literature, over the course of 20 years or more. This is far from the only instance. And this is an especially easy story to verify (or not).

Seguin, E. (1870). Art. XXXIII.-New Facts and Remarks concerning Idiocy: being a Lecture delivered before the New York Medical Journal Association, October 15, 1869 American Journal of the Medical Sciences, 59 (129), 518-519

The press release starts by declaring that autistic children "lack visual skills required for independence" and does not exaggerate the claims in the paper, which merit a lot of scrutiny. So bear with me, this is not going to be short. First what they did (and didn't do), then what they found, then what it means.

1. What did they do? And what didn't they do?

Pellicano et al.'s purpose was to assess systemizing in autism by testing autistic visual search abilities in true-to-life large-scale space. And this research group has the set-up for it--a room-size visual search task. Except they ignored this possibility, instead choosing what they themselves call a test of "non-visually guided foraging."

In other words, Pellicano et al. do not include visual search tasks at any scale, so you can't draw conclusions from their paper about visual search abilities in autism.

It took place in a 4m by 4m space arranged as shown. In two 20-trial blocks, 20 autistic and 20 nonautistic children (age ~11yrs) had to find which of 16 green floor lights, eight on each side of a midline, turned red when pressed. The children were not told that 80% of the concealed red targets were on one side of the room ("rich" side) with only 20% on the other ("sparse" side).

The task instruction was to find the red target as quickly as possible.

But search all you like, in this paper you will find no data about how quickly the participants found the targets. The researchers left out this available measure. They left out number of green lights hit before finding the red target. They left out how many first touches were on the "rich" vs "sparse" sides of the room.

They left out almost every measure they previously deemed important to assess performance on foraging tasks in large-scale space (e.g. here, here, here).

They seem also to overlook their previous systemizing-relevant (you would think) finding of no male-female difference in performance on the same foraging task (experiment 2 here).

Another of their own previous findings is that children's foraging performance is related to their performance on the Corsi block task, a measure of spatial memory. Autistic children don't necessarily show a deficit on this task (e.g. here), but in Pellicano et al., they did. If the goal wasn't simply to assess spatial memory, why not match groups on this ability?

2. What did they find?

Their main measure was sensitivity to probability in the foraging task which, ignoring their previous work, they defined as percent of visits to the rich side of the room. Here the authors report "no overall effect of group," never mind media reports (here is one). There was an effect for the first block of trials, where autistics visited the rich side for 45.50% of their touches vs 61.50% for nonautistics. In the 2nd block, the figures were 62.25% and 63.25%.

Two additional novel foraging measures involved total length of paths to the red target, per floor distance between green lights. These were assessed with highly sophisticated but conspicuously small-scale methods suited to eye-scanning and/or non-foraging pencil-and-paper tasks. So it doesn't matter how much time you take, or how much you have to twist and turn, or how many green lights you have to reach for. All that matters is length of path, and at this level the autistic children were judged significantly (a) less optimal and (b) less repetitive. Because you should ask, according to Pellicano et al., less repetitive means less systematic ergo is now yet another autistic deficit.

The final foraging measure was total revisits to green lights, where the autistics had significantly more in the second but not the first block. One autistic child racked up a big chunk of the autistic revisits (more than 3 SDs above the group mean), which might indicate an atypical understanding of or interest in aspects of the task. There is no information about group differences minus the interesting outlier.

The two non-foraging measures were the Corsi task where autistic children had a significant disadvantage, and an embedded figures task on which they performed significantly better than their controls (yes, interpreted as a global deficit, without evidence for same). Revisits--outlier included--correlated with the non-foraging measures in the autistic group. There is no information about these correlations for the nonautistic children or about any other correlations.

3. What does it mean?

Well first, it's an interesting task, even if it's not a visual search task.

But even if autistics totally failed (they didn't, and search all you want again, but you will find no rationale in this paper for the drop-off-a-cliff thresholds pushed by the authors), this task doesn't map easily onto the authors' sensational claims. These include that autistics can't find "shoes in the bedroom, apples in a supermarket, or a favourite animal at the zoo" ergo can't achieve independence.

Of course I want a whole lot more data, or an excellent rationale (none is provided) for not reporting most of it. And numerous possibilities were overlooked.

There's those other regularities in the distribution of red targets. There were only 40 trials, and each green light position was the target at least once. On the "sparse" side, each position would be the target exactly once (8 targets, 8 positions). The participants were not supposed to notice or be affected by this kind of regularity.

No one knows how autistics would have performed if given accurate task instructions (to take the shortest path, as measured by the authors, to the target). Maybe someone else can bring up motor differences, which plausibly are relevant to this "true-to-life" task. And I wonder how clear, for autistics, the task instructions were with respect to revisits.

Autistics should be notorious by now for noticing aspects of tasks that nonautistics don't (fantastic example at IMFAR last year), and for exploring more possibilities than nonautistics (examples here and here). Writing this off as a bad thing, as autistics being lost in space or some dire equivalent, is shortsighted to say the least.

If you're paying attention, autistics' "less repetitive" and speculatively more exploratory behavior--more aspects of the array are interesting to autistics, more possibilities and probabilities are explored--in "large-scale" space is a predictable finding. There is no reason to trash as useless (or harmful, and Pellicano et al. go there) multiple replications of excellent autistic visual search and probabilistic learning performance. One genuine conclusion is that systemizing, as conceived by Pellicano et al., and also by Baron-Cohen, is too rigid and limited a model to encompass how autistics atypically detect and explore environmental regularities.

Reference:

Pellicano E, Smith AD, Cristino F, Hood BM, Briscoe J, & Gilchrist ID (2010). Children with autism are neither systematic nor optimal foragers. Proceedings of the National Academy of Sciences of the United States of America PMID: 21173235

Postscript: You can find a large-scale space, true-to-life, real-world (and wonderful) embedded figures task performed by an autistic child here.

Strange way to start the year. And maybe this recent search from the University of Southampton is actually unrelated to the university. On the other hand, in the current era of autism politics, it seems plausible to wonder whether this is part of someone's assignment or coursework or whatever.

And if it is, is the question being asked about all autism researchers, or only autistic ones?

In the scientific literature, you can find powerful deficit models of autism at work in predictions that autistics should disproportionately be violent and prone to criminal behavior. For decades now, examples and claims (just a few here, here, here, here) fitting this prediction have been highlighted, while the few dissenting views (e.g. here and here) have had little effect.

Then there's political expedience. Unless we receive the usual lobbied-for interventions, autistics will disproportionately display criminal behavior and wind up in jail--or so it is claimed. A few examples from the usual autism politics here, here, here.

Kathrin Hippler and her colleagues deserve high praise for noticing how well-placed they were to investigate these kinds of popular claims. Their recent paper takes advantage of their access to information about the large group of individuals--177 of them--who are former patients from Hans Asperger's clinical practice in Vienna.

Most (93%) of these former patients are male. All were assumed to score at least in the normal range of intelligence as children, but for most there are no recorded IQ scores. They were born between 1938 and 1979, and on average were diagnosed at age eight (range 3-21 years). In 2010 their average age would be about 50. It is unlikely that most of this cohort would have undergone the usual lobbied-for autism interventions as children.

Hippler et al. obtained information from the Austrian Penal Register about all criminal convictions registered, as of 2002, in this cohort. They found 33 convictions for a total of 8 individuals, resulting in 23 "custodial sentences" ranging from 2-30 months and 11 fines.

They also checked whether, compared to the general population, their cohort had a higher rate of newly-registered convictions for the years 1998-2002. Here is what they found:

the average proportion of convictions found in our sample (1.30%) is very comparable to that in the general male population (1.25%)

As to kinds of crimes:

By far the most common convictions in Asperger’s former patients were for property offences [...] Offences against life and physical integrity were rare.

And while data for the general population were limited:

qualitative assessment of offence types in Asperger’s former patients suggests that they do not differ radically from those in the general public

Because this point needs to be underlined, here is more from Hippler et al.'s discussion:

the findings from our study do not suggest an over-representation of certain offence types. In the case records spanning 22 years and 33 convictions, there were only three cases of bodily injury, one case of robbery and one case of violent and threatening behaviour.

Again to their great credit, Hippler et al. also provide data broken down according to Asperger's system of diagnosis. The 177 former patients were divided into an AP group ("autistic psychopathology," N = 73) and an AF group ("features of autistic psychopathology," N = 104).

Some guesswork is involved, but the AP group would fall under current criteria either for Asperger syndrome or the specific diagnosis of autism. Hippler et al. conservatively estimate one-third would be specific-autistic, but it's not difficult to find researchers who would guess a higher proportion (here, for example).

While a minority of the AF group might meet Asperger syndrome criteria, according to Hippler et al., others might be PDD-NOS, or in the (nonautistic) broader autistic phenotype. The AFs are described as "former patients at the less extreme end of the spectrum" and were included by Hippler et al. because:

Asperger believed that ‘autistic psychopathy’ was a heritable condition blending into "normality", which is reflected in the case descriptions of these children in the sense that the core features were the same but symptoms were less severe or could be compensated for better.

According to near-universal assumptions, it would be far better to be AF (less "severe" or "extreme") than AP. Transforming AP-types into AF-types is a major goal of the usual lobbied-for autism interventions. But Hippler et al. found that most of the registered criminal convictions in their cohort belonged not to the "more autistic" AP group but to the "less autistic or not autistic at all" AF group.

Indeed, of the 33 convictions found registered as of 2002, only three convictions of two individuals were found in the AP group. The remaining 30 belong to six individuals in the AF group, with two in this group contributing 22 convictions.

In the comparison with rates of newly-registered convictions in the general population, the AP rate was 0.6% while the AF rate was 1.7%. These figures are lower and higher, respectively, than for the general male population, while the AP rate is comparable to the general population, females included (0.7%).

Hippler et al. provide a competent overview of the relevant literature (including this recent finding), as well as a fair discussion of their study's limitations. Under the banner "Wider Implications," they write:

There is a public perception that individuals with mental health diagnoses in general, and Asperger’s syndrome in particular, present a threat to the general public. We contend that, based on the follow-up data from Asperger’s original cohort, as well as other studies, this perception is wrong.

Even wider implications include the neglected question of how being regarded as just naturally violent and dangerous to others, as natural born criminals, has affected the outcomes of autistics.

Tuesday, August 31, 2010

If you are autistic and ever venture or are pushed into public, a near-certainty is that you will publicly be ranked and classified by total strangers.

For example, you will be assigned to the "high end" or the "low end" of the autistic spectrum, according to whether you are claimed to have a good or bad outcome (I've been claimed to have both). Non-political observers may notice how ethically and scientifically problematic this is, but there are few discussions, formal or informal, in which autistics aren't automatically assigned "high-functioning" and "low-functioning" rankings as definitive and permanent aspects of ourselves: there are LFAs and then there are HFAs.

If the whole area of autism were not so politicized, it might have long been seen as important to check the scientific basis for these ubiquitous rankings. It might even be seen as an ethical obligation. It might be noticed that there is something wrong with discarding and therefore dishonouring the contributions to research made by the countless autistic children and adults who have been recruited as participants in autism research, and without whom there would be no autism research at all.

I've never been impressed by autism politics, on whatever "side," so I'll start with some basic information about levels of functioning as reported in autism research, then provide several examples pulled from some of the numerous papers I've recently looked at.

What does level of functioning mean in autism research?

In autism research, autistics' level of functioning is most often judged according to scores on specific tests of IQ (e.g., Wechsler) or developmental level (Mullen, Bayley, sometimes the Vineland) at a specific time.

The reported threshold dividing "high" from "low" functioning ranges from 50 to 90--at least in papers I've read so far; the actual range might be even greater. Those are IQ or IQ-type scores with a mean of 100 and a standard deviation of 15. So the threshold, the line dividing "high" from "low" functioning in autism research, is almost three SDs wide. Fall into that impressive span, and you may be high or you may be low functioning, depending on who you ask.

If 90 is the threshold, then about 25% of the entire population (autistic, nonautistic, everyone) is low-functioning. If the more common threshold of 85 is chosen, then about 16% of everyone is low-functioning.

70 is the threshold often considered to be standard even if in reality, if you read the literature, the threshold varies dramatically. But there are different tests and within commonly-used tests there are different ways to set a threshold, even when the threshold is numerically set at 70.

With this in mind, here are some examples.

Example 1

In Farley et al. (2010) the threshold is indeed 70 and autistics with lower baseline (circa age 8yrs) IQs were excluded. As the authors write, "all participants had baseline IQs in the nonimpaired range."

In fact the full-scale baseline IQs of autistics deemed to meet the 70 threshold, and included as participants, range down to 36. An IQ of 36 is near the current threshold for severe intellectual disability. An IQ of 36 (if you were under age ~4yrs) would exclude you from Ivar Lovaas' famous ABA study--you are too low-functioning.

But in different circumstances, which are specified by Farley et al., a full-scale IQ of 36 can also be considered to grossly underestimate your abilities and to put you in a study of participants who would widely be ranked as high-functioning.

Example 2

Klin et al. (2007) is another study where the autistic participants (ages 7-18yrs) are all considered high-functioning and the threshold is 70. In one of two samples, verbal, performance, and full-scale IQs run from 52 to 150. Then there are Vineland scores. While Vineland is a test of adaptive behavior, in autism research Vineland scores can also be used as measures of intelligence ergo level of functioning.

Vineland composite scores in this sample range down to 25. Two of the Vineland domain scores range down to 20. In a recent population-based study (ages 9-14yrs), an assigned Vineland score of 19 was used to represent profound intellectual disability. But in Klin et al., you can have a Vineland composite score of 25, or Vineland domain scores of 20, and you are high-functioning. You are in the same category as someone with a verbal IQ of 150.

Example 3

In Akshoomoff et al. (2004), there is also a threshold of 70, but autistics falling on either side of this threshold are included then divided into lower and higher functioning groups. Performance IQ scores in the lower-functioning group range up to 128. That is not only higher than the individual scores of the entire higher-functioning group, it is higher than all but ~3% of the entire population. If a PIQ of 128 ranks you as low-functioning, as it does in Akshoomoff et al., then almost everyone is low-functioning.

Example 4

In view of the above it might seem wise to abandon the 70 threshold and try something completely different. Annaz et al. (2009) created high-functioning and low-functioning autism groups (ages 5-11yrs) by incorrectly using the CARS (Childhood Autism Rating Scale), which is supposed to be a measure of autism "severity." This study features two measures of intelligence, one verbal (British Picture Vocabulary Scale, a verbal IQ equivalent) and one non-verbal (a subtest from the British Ability Scales).

One result is that you can have a verbal IQ of 62 and be classified as high-functioning, and a verbal IQ of 111 and be classified as low-functioning.

The non-verbal measure is reported in age-equivalents only, but the two autistic groups are remarkably well-matched on age. And the mean non-verbal age equivalents are both (1) the same for the two autistic groups, and (2) very nearly the same as mean chronological age, again for the two autistic groups.

In other words, another result is that both high- and low-functioning autistics here represent groups whose mean measured non-verbal intelligence is the same as the general population mean, and of course vice-versa. Setting aside possible differences in distribution, now everyone is both high- and low-functioning.

Conditional examples 5 and 6

This is a bit of a digression, but if you adhere to the common political or ideological prejudice that "Kanner's autism" is "classical autism" is "low-functioning autism" you are then required to achieve an IQ of over 140 (from Kanner's original 11) or 150 (from Kanner's 1956 follow-up) to be ranked as high-functioning. That means you have to be better than the 99th percentile. Now what?

The above doesn't nearly convey the arbitrariness in the existing autism literature. As I wrote, I've provided just a bunch of examples, among many others, you will find if you read a lot of papers. Also, I've left out entire major areas, like changes over time and results from deliberate comparisons between different tests of intelligence.

While this was not intentional, the examples above might be construed as exploiting the atypically high variability characterizing individual autistics and in addition characterizing autistics as a group. Then the question is whether it serves the interests of autistics, and whether it advances autism research, to diminish, misrepresent, trivialize, denigrate, obscure, or deny this characteristic variability.

And as usual, the above can be verified by reading the existing autism literature. If there are any factual errors, as is always possible, please let me know.

Futher reading

The late Ivar Lovaas expressed his views about levels of functioning in autism in one of the major ABA manuals; this is quoted here. From a different point of view, this article concisely applies ethical consideration to the issue of level of functioning, among others. In autism politics, the dimension of autism "severity" and the different dimension of "level of functioning" are often wrongly confused or conflated. You can find science-based information about autism "severity" here and here.

Wednesday, August 18, 2010

Some months ago, Jonathan Green and his colleagues simultaneously published (in the Lancet) and presented (at IMFAR 2010) their multi-site RCT of an early autism intervention. In conducting and reporting the Preschool Autism Communication Trial, they have made autism research history. They have done so simply by applying to autistics scientific standards that are well-established in non-autism non-ABA areas. The upshot is an autism early intervention trial whose results are worth serious consideration and which is unprecedented in this respect.

Via the PACT website, you can find the Lancet paper reporting the PACT RCT, the peer-reviewed PACT intervention "manual" (which refers to another, more detailed manual), the PACT RCT protocols, references for related papers including a 2004 pilot RCT, a list of investigators and funders, and so on.

I've previously written about the PACT as an anomaly in autism intervention research. The PACT entails an early autism intervention that was not widely promoted as effective or essential before it was fairly tested. That is a first in the history of autism research. It may also be the largest RCT of any kind of autism intervention ever published (if I'm wrong, I'm sure someone will tell me).

In short the PACT RCT is an internationally-registered multi-site trial of a manualized intervention targeting the parents of preschool (ages 2-5yrs) autistic children. The PACT intervention spans one year and involves 12 twice-monthly 2hr clinic-based sessions followed by 6 monthly sessions. Parents are expected to apply what they learn, in interacting with their autistic child, for half an hour per day.

For the PACT RCT, 152 children who met criteria for the specific diagnosis of autism were randomly assigned to receive either the PACT intervention plus treatment as usual or TAU alone. PACT and TAU children were well-matched at intake across several measures, and finished well-matched in other treatments received. Parents were not as well-matched. A detailed description of the PACT intervention is here.

Dr Green and colleagues were successful both in recruiting an unprecedentedly large number of preschool autistic children for an RCT--in fact, expectations for recruitment were exceeded--and in minimizing drop-outs. In the Lancet, you will find an outstandingly clear CONSORT flowchart of participants as well as an intention to treat analysis and mixed results.

Results favouring the PACT group were mainly in the area of parent-child interaction, as assessed through videos. The PACT group also outperformed the TAU children in parent-reported vocabulary and social measures, results carefully downplayed by Green et al. due to the parents not being blinded to intervention status. Post-treatment-only measures of adaptive behaviour reported by teachers did not differ between groups.

The main measure was the ADOS used as a scale, and here too there was no result. In my view, using the ADOS as the main measure is a major error but an informative one. The ADOS, as used here (awkward tweaks and all), has thresholds only and doesn't work like a scale. And where is the evidence that lower ADOS scores in very young autistic children in themselves lead to better eventual outcomes? On the other hand, the strong design of the PACT RCT is such that it calls into question what exactly the ADOS is assessing--a result which deserves a lot of attention.

Using ADOS thresholds, 27% of the 146 children who completed their assigned treatment had migrated from the specific diagnosis of autism to the lower-threshold "ASD" category, while an additional 6% no longer met criteria for any autistic spectrum diagnosis. No group differences were found. These findings lend perspective to the widely-publicized diagnostic category results from another recent RCT involving a small sample of much younger children receiving more than two years of intervention.

As with all studies, it's possible to list a range of flaws, potential concerns, and caveats. The ADOS is not only wrongly used as a measure, it is incorrectly used to stratify the sample for randomization. The ADI-R is used in a slightly unconventional way, possibly to accommodate children who otherwise would be excluded by its limitations. There was no screening ergo no exclusion for genetic syndromes, which arguably makes the findings more difficult to interpret. There was poor agreement between raters for two of the three parent-child interaction measures, which in my view is fascinating and telling: nonautistic raters are not good at agreeing about what child initiation and shared attention consist of, when a child is autistic. If you look at the Lancet paper, depending on how your brain works, you should rapidly find at least one minor error in the data (I can't help it...).

The PACT intervention itself has questionable aspects. The fundamental idea is sound: train parents to better respond to their autistic children's communication. But the PACT is a rigidly developmental six-step approach based on how nonautistics develop. In addition, and like so many other autism interventions, the PACT imposes extreme rationing of information and materials. Everything is cleared and shut away except a few toys or items. Anything in which autistics regardless manage to show strong interest will be taken away and replaced with something less interesting. What this does to an autistic child's communication and learning is not considered. The PACT explicitly requires every effort to identify and cater to the parents' different ways of learning, but no such consideration is extended to the autistic children.

The above is a not-close-to-complete list of the PACT's strengths, many of them unique, and its weaknesses--which due to its strengths serve as important information for the future of autism intervention research. Because the trial is so strongly designed compared to the rest of the literature, it is that much more important to learn from and build on. This may not serve the interests of various lobby groups or service providers or political leaders or advocates, but it will serve the interests of autistics. Here is a suggestion from Dr Green and colleagues in the Lancet:

These findings suggest that the optimistic results from other studies should be reassessed.

Yes--good experimental design means that you risk not having your biases confirmed. But taking this risk is currently the only demonstrated way to fairly test interventions and treatments, and for this reason, the only way to conduct ethical research.

Monday, May 17, 2010

I had no idea my sleep-wake cycle was pathological until I saw a presentation a few years ago by the geneticist Thomas Bourgeron. One of his interests is clock genes in autism. In fact only by speaking with him did it dawn on me that I lack a circadian rhythm.

As it turns out, autistics are considered to have circadian clocks "gone bad." Two reviews (Bourgeron, 2007; Glickman, 2010) cover the evidence with respect to autism and circadian rhythms, most of it in the direction of comprehensive atypicalities. Glickman (2010) summarizes the problem:

Disturbed sleep-wake patterns and abnormal hormone profiles in children with autism suggest an underlying impairment of the circadian timing system.

And autistic children's bad timing, when it comes to sleeping and waking, is considered disruptive or worse, both to themselves and their sleep-deprived families. Bourgeron (2007) raises the further concern that typical sleep-wake patterns are essential for "appropriate" development, for the functioning of memory, and so on. Apparently there is nothing good about autistics' bad circadian clocks.

Autistics whose sleep-wake cycles carry on independently from environmental and social cues are said to be "freerunning." My form of freerunning is extreme. I can sleep whenever I want--a few hours here, twelve hours there, as necessary, no matter the time of day or night. I enjoy sunlight but its absence has no effect on my mood, or on my sleep. I can work through nights, no problem, and shift to days in a blink. I've never suffered through jet lag. And so on. Until I ran into Bourgeron I had no idea this was considered pathological, at least in an autistic.

The usual response to freerunning in autism is to see this as an autism-related sleep disorder. There is very preliminary evidence that freerunning autistics can be successfully treated with melatonin. Bourgeron (2007) refers to a short case study about an autistic whose free-running was remediated by melatonin treatment.

Of course nonautistics take melatonin too. They put themselves through sleep hygiene routines and they go to sleep clinics in droves. They suffer terribly from shift work and jet lag, and inadequate efforts to eradicate this suffering are everywhere, from new drugs to new iPhone apps.

There is an enormous market for products and services that can treat individuals who stray from the proper expected sleep-wake cycle. Remarkably, the goal is never to free people from their circadian prisons but to return them there and keep them there. The schedule may be changed, with effort, but the prison remains the same.

The next time I spoke with Bourgeron, I told him there should be a sort of anti-melatonin which would allow people to freerun like me, ergo avoiding the ills of jet lag, shift work, and what-all. It seems possible no one has seriously thought of this.

I suggested the anti-melatonin idea to some nonautistic colleagues. They burst into stories about destroyed careers, disrupted lives. In constrast with autistics like me, my colleagues have perfectly "good" circadian clocks--which turn out to be incompatible with their challenging lives. They suffered, for being normally chained to their normal circadian rhythms.

If there was a pill that could give them my very dysfunctional clock so they could freerun to the extreme, they would buy it in bulk. But there isn't. Instead there is the assumption that everyone has to live in a circadian prison.

Glickman (2010) speculates that some autistics' failure to chain our sleep-wake cycles to environmental cues (you can find information about entrainment, as this process is called, here) may arise from our atypical perception. My totally wild guess might be that an extreme freerunning phenotype in autism may be contributed to in part by cognitive versatility in autism, which would result in perceived environmental cues affecting sleep-wake cycles in an optional rather than mandatory way.

The renowned Bora Zivkovic, who unlike me knows all about circadian clocks, recently wrote that--unlike me--typical people have "have complex circadian systems that are easy to get out of whack" with dire consequences:

In the state of almost permanent jet-lag that many of us live in, a lot of things go wrong. We get sleeping disorders, eating disorders, obesity, compromised immunity leading to cancer, problems with reproduction, increase in psychiatric problems, the Seasonal Affective Disorder, prevalence of stomach ulcers and breast cancer in night-shift nurses, etc.

BoraZ was writing enviously about reindeer who live with drastic changes of season yet have escaped this kind of misery, possibly by having a "low-amplitude" clock. They just stroll out of their circadian prisons, whenever it's adapative. How can poor suffering human beings do this, is the question--finally. It's a very good question.

The heart of this study is a computer game version of catch which appears to involve four human players. When a player is thrown the ball, he must then throw it to another player of his choice. Every time a player receives the ball, he receives a bit of money.

In Andari et al. (in press), small groups of autistic and nonautistic adults ("P") individually play this game with three strangers ("A" "B" "C"). Much is done to persuade the autistic and nonautistic participants that the strangers, who in fact are elaborately programmed, are actual, present, proximate (in adjacent booths), decision-making human beings.

The three strangers start by equally distributing the ball to the other players. Then the elaborate program kicks in: A and C increasingly favour one player (P and B, respectively) while shunning the others. B's behaviour does not change.

In a prototypical display of us-vs-them thinking, the nonautistic Ps responded by ganging up with A to reap the social and monetary gains of a close alliance founded on the exclusion of half the players.

The autistic Ps in contrast displayed no such selfish and discriminatory behaviour. They continued to throw the ball to the other players in equal proportion, ignoring their self-interest in favour of keeping all players equally included.

Further, subjective ratings of the other players revealed that the autistic Ps did not have the kinds of biases that are routinely called hypocrisy. They did not judge C, who shared the most with B, as worse or less trustworthy than A, who shared the most with themselves.

According to Andari et al. (in press), autistics "cannot understand or engage in social situations," as evidenced by autistics' outstandingly altruistic performance in this game. It is this profound social deficit, this altruistic autistic behaviour, that was targeted for treatment.

And indeed, the treatment was successful. Autistics randomly administered a nasal mist containing oxytocin, rather than a saline placebo, significantly improved. They became willing to work with one of the players in an effort to shun and discriminate against the other two, and thereby get more than their fair share of money and attention. They became willing to see the player who shared with them as good and trustworthy, and the player who shared with someone else as bad and untrustworthy. They learned and displayed selfishness and hypocrisy and us-vs-them thinking. Their objectivity, fairness, and altruism were--temporarily--cured.

Then this finding was replicated in a second small group of autistic adults who performed the same task but without monetary rewards.

Success! Cue the avalanche of blogging and media stories. Uta Frith says, "This could be revolutionary."

There is more to Andari et al. (in press), which also reports on two tasks involving face images, about which a few quick notes:

1. There is little ecological evidence that autistics avoid face images or do not make eye contact with face images.

2. Numerous other studies (a few examples here, here, here, here) which feature stronger designs (e.g., use of fixation crosses) have found typical duration and distribution of visual fixations to human face images, and/or typical attention to the eye area, in autistics.

3. For the nonautistic participants, the reported total visual fixation time to the face images--the crucial measure--well exceeds the total exposure time (both tasks), which has me, for one, scratching my head.

4. As yet there is no evidence that the reported oxytocin-related measured changes in visual fixation times to face images would overall be more beneficial than harmful to autistics.

5. In other studies (examples here and here) faster saccades and shorter visual fixation times, presumed by Andari et al. (in press) to be a very bad thing, have been associated with enhanced performance in autistics.

All this too is telling in its way, but it cannot equal the reported spectacular findings with respect to autism and altruism. In this respect Andari et al. (in press), even with its obvious limitations in design, is an enormously valuable paper. It has much to reveal about how autistics are regarded, and about the nature and consequences of the fight against autism.

Friday, February 12, 2010

We have to wait, patiently, for the DSM-V people to cough up their system for ranking and classifying all autistics according autism "severity." In the meantime, some recently reported data are worth mulling over.

First, here is the most recent unofficial DSM-V autism "severity" ranking-system proposal, and here is my response, including information about instruments commonly claimed to measure autism "severity." The current official DSM-V void in this area can be located here.

An increasingly prominent measure of autism "severity" is the Social Responsiveness Scale. Developed by John Constantino, it now exists in different age-range versions. Its purpose encompasses quantifying what are presumed to be autistic traits, from none on up, across the full range of humanity.

The SRS is a 65-item questionaire most often filled out by parents or teachers, whose ratings classify children according to "severity of autistic symptomology." Higher scores, above an established threshold, indicate greater autism "severity," and are therefore considered worse.

In a recently epublished high-profile paper (Roberts et al., in press), SRS scores were reported for 25 autistic children who were recruited then divided into two groups according to performance on a popular test of language abilities.

The children were about 10 years old. Sixteen autistic children classified as non-language-impaired achieved an average language ability score of ~100, right at the mean for the administered test. In contrast, nine autistic children classified as language-impaired scored ~65, more than two standard deviations lower.

I'm going to ignore the main purpose of this interesting study, which used magnetoencephalography to compare auditory evoked responses in autistics and nonautistics. Instead I'll concentrate on how this divided sample of autistic children was characterized.

Apart from language scores, the authors reported what could be considered verbal and performance intelligence, as per indexes from the latest child version of the Wechsler scales. Here the non-language-impaired autistic children scored significantly higher, with ~20-point and ~16-point advantages, respectively, over the language-impaired autistic group.

As I wrote above, Roberts et al. (in press) also reported SRS scores, described here as "dimensional symptom severity ratings."

SRS raw scores are commonly used in research, but the SRS has also been standardized, providing T-scores which account for variables such as gender and differences between raters (e.g. parents vs teachers). T-scores are standard scores with a mean of 50 and a standard deviation of 10.

While the SRS is not yet considered a diagnostic instrument, a T-score of 60, one SD above the mean, is currently the SRS-based cut-off for an autistic spectrum diagnosis. Standard scores from 60 to 75 currently indicate SRS-based "mild to moderate" autism; 76 or higher is the SRS-based "severe" autism range.

As might be predicted, the two autistic groups in Roberts et al. (in press) differed significantly on SRS T-scores; that is, they differed on autism "severity." One group scored on average 70.89, falling into the "mild to moderate" range. The other scored 81.44, a full standard deviation higher, and crossed the threshold into "severe" autism.

As wouldn't be predicted at all, the non-language-impaired autistics, who had advantages not only in language but on measures of intelligence, were the "severe" autistics, while the language-impaired children were the "mild to moderate" ones.

My wild guess is that if an autism intervention was shown in fair tests to consistently produce a reduction in SRS scores of a full standard deviation, and what's more, transported groups--however small--of autistic children from the "severe" to the "mild to moderate" SRS range, this would be considered tremendous progress.

The sample of autistic children in Roberts et al. (in press) is of course very small, as is charateristic of the bulk of the autism literature, and much can happen within such small sample. But these data aren't inconsistent with numerous other existing reports, and deserve at least a look while we wait, patiently, for the DSM-V people to proclaim on autism "severity."

You can find an uncritical short blurb about the SRS, which used to be called the "Social Reciprocity Scale," here, and a more extensive, critical description here.

Many thanks to Jennifer Stevenson for (patiently) answering my questions about the SRS. Any factual errors are entirely mine and should you find any, you should alert me immediately.